Literature DB >> 25863307

Molecular pathogenesis of Marfan syndrome.

Chrishan J A Ramachandra1, Ashish Mehta1, Kenneth Wei Qiang Guo2, Philip Wong3, Ju Le Tan2, Winston Shim4.   

Abstract

Marfan syndrome (MFS) is a genetic disorder that affects multiple organs. Mortality imposed by aortic aneurysm and dissections represent the most serious clinical manifestation of MFS. Progressive pathological aortic root enlargement as the result of degeneration of microfibril architecture and consequential loss of extracellular matrix integrity due to fibrillin-1 (FBN1) mutations are commonly diagnosed clinical manifestations of MFS. However, overlapping clinical manifestations with other aneurysmal disorders present a significant challenge in early and accurate diagnosis of MFS. While FBN1 mutations, abnormal transforming growth factor-β signaling and dysregulated matrix metalloproteinases have been implicated in MFS, clinically accepted risk-stratifying biomarkers have yet to be reliably identified. In this review, we summarize current consensus and recent insights in the understanding of MFS pathogenesis. Finally, we introduce the application of induced pluripotent stem cells (iPSCs) as cellular models for MFS and its potential as a novel platform into providing better appreciation of mechanisms underlying MFS diverse manifestations in the cardiovascular system.
Copyright © 2015. Published by Elsevier Ireland Ltd.

Entities:  

Keywords:  Aortic aneurysms; Fibrillin-1; Induced pluripotent stem cells; Marfan syndrome; TGF-β

Mesh:

Substances:

Year:  2015        PMID: 25863307     DOI: 10.1016/j.ijcard.2015.03.423

Source DB:  PubMed          Journal:  Int J Cardiol        ISSN: 0167-5273            Impact factor:   4.164


  23 in total

1.  Identification of FBN1 gene mutations in Ukrainian Marfan syndrome patients.

Authors:  Rustam Zhurayev; Dorien Proost; Dmytro Zerbino; Viktor Fedorenko; Josephina A N Meester; Lut VAN Laer; Bart L Loeys
Journal:  Genet Res (Camb)       Date:  2016-10-11       Impact factor: 1.588

Review 2.  The influence of genetics in congenital diaphragmatic hernia.

Authors:  Lan Yu; Rebecca R Hernan; Julia Wynn; Wendy K Chung
Journal:  Semin Perinatol       Date:  2019-08-01       Impact factor: 3.300

3.  Panlobular Emphysema: Enhancing Visibility with Quantitative Computed Tomography.

Authors:  Surya P Bhatt
Journal:  Chronic Obstr Pulm Dis       Date:  2016-05-23

Review 4.  How can genetic diagnosis inform the decision of when to operate?

Authors:  Maral Ouzounian; Scott A LeMaire
Journal:  J Vis Surg       Date:  2018-04-03

Review 5.  Molecular targets in aortic aneurysm for establishing novel management paradigms.

Authors:  Chengkai Hu; Kai Zhu; Jun Li; Chunsheng Wang; Lao Lai
Journal:  J Thorac Dis       Date:  2017-11       Impact factor: 2.895

6.  Fibroblast-Secreted Phosphoprotein 1 Mediates Extracellular Matrix Deposition and Inhibits Smooth Muscle Cell Contractility in Marfan Syndrome Aortic Aneurysm.

Authors:  Mei Zhou; Yuexin Zhu; Zeyi Zhou; Feiran Qi; Shuai Zheng; Shijuan Gao; Yulin Li; Yan Liu; Jie Du
Journal:  J Cardiovasc Transl Res       Date:  2022-04-12       Impact factor: 4.132

Review 7.  Open Versus Endovascular or Hybrid Thoracic Aortic Aneurysm Repair.

Authors:  Ryan Clare; Julianne Jorgensen; Somjot S Brar
Journal:  Curr Atheroscler Rep       Date:  2016-10       Impact factor: 5.113

8.  Two novel mutations of FBN1 in Jordanian patients with Marfan syndrome.

Authors:  Saied A Jaradat; Lama A Abujamous; Ali A Al-Hawamdeh; Khaldoon M Alawneh; Tamara A Rawashdeh; Zaher M Jaradat
Journal:  Int J Clin Exp Med       Date:  2015-10-15

Review 9.  Skeletal manifestations of Marfan syndrome associated to heterozygous R2726W FBN1 variant: sibling case report and literature review.

Authors:  Octavio D Reyes-Hernández; Carmen Palacios-Reyes; Sonia Chávez-Ocaña; Enoc M Cortés-Malagón; Patricia Garcia Alonso-Themann; Víctor Ramos-Cano; Julián Ramírez-Bello; Mónica Sierra-Martínez
Journal:  BMC Musculoskelet Disord       Date:  2016-02-15       Impact factor: 2.362

Review 10.  The Molecular Genetics of Marfan Syndrome.

Authors:  Qiu Du; Dingding Zhang; Yue Zhuang; Qiongrong Xia; Taishen Wen; Haiping Jia
Journal:  Int J Med Sci       Date:  2021-05-27       Impact factor: 3.738

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